Discussion

BD is a form of intraepidermal squamous cell carcinoma in situ with a small potential of invasive malignancy.[1] It may affect both skin and/or mucosa. The incidence is around 1.42 per 1000 population.[3] BD is uncommon among type V Fitzpatrick skin people.[4],[5] However, we have reported these five cases within 1 year, indicating their increased prevalence among Indian skin.

It can occur at any age but mostly seen in the elderly having equal sex preponderance. It usually occurs as a solitary lesion over sun-exposed areas.[5] Multiple lesions are seen in 10%–20% of cases with history of arsenic exposure. Case 5 had multiple lesions over both uncovered and covered areas without any history of arsenic exposure.[2]

The common sites for BD include the chronically photo-exposed sites such as head and neck and dorsum of hands and lower legs.[5],[6] Its occurrence in photo-covered sites is rare. In our case series, all patients presented with lesions on unusual sites. Our first case has lesion over the finger, the second case over the back, and the third and fourth cases presented over the penis and thigh, respectively. To the best of the author's knowledge, there was only one case report of BD presenting over the finger by Singh et al.[1] Similarly, one case reported over the penis by Dandale et al.[7] All our cases were misdiagnosed previously and presented late.

Clinically, BD presents as a solitary well-demarcated, erythematous, crusted, or scaly plaque.[1] This type of morphology may be commonly confused with psoriasis, nummular eczema, actinic keratosis, bowenoid papulosis, and discoid lupus erythematosus. Other uncommon clinical variants include verrucous, hyperkeratotic, atrophic, and pigmented types.[1],[8] Among them, the pigmented variant is still very rare with <2 href="https://www.thieme-connect.com/products/ejournals/html/10.4103/ijmpo.ijmpo_366_20#BR_5" xss=removed>5],[9],[10] In the present series, two cases presented with solitary pigmented lesion and one case presented as widespread pigmented lesions. As per the author's knowledge, there is only one case report of widespread pigmented BD by Gahalaut et al.[2] Furthermore, we could not explain widespread distribution in our case which is usually described with arsenic exposure. The occurrence of widespread lesions in an immunocompetent individual has not been reported previously.

BD occurs on the skin damaged due to chronic ultraviolet radiation exposure, human papillomavirus infection, arsenic exposure, previous radiation, immunosuppression, trauma, and genetic factors.[3] However, in all our cases, we did not find any predisposing factor to be correlated.

The diagnosis of BD is based on clinical suspicion and histopathology. Histological examination of BD reveals hyperkeratotic epidermis with a variable degree of parakeratosis. The cells throughout the epidermis lie in complete disorder resulting in a “windblown appearance” showing atypia with large hyperchromatic nuclei with intact basement membrane.[1] In all our cases, we found cellular disarray and atypia with intact basement membrane, indicating that there was no progression toward squamous cell carcinoma.

Various treatment modalities used are topical imiquimod cream, topical 5-fluorouracil cream, cryotherapy, surgical excision, curettage and electrocautery and photodynamic therapy, lasers, and topical diclofenac.[11] They were successfully treated with topical 5% imiquimod cream in three cases and surgical excision in two cases.

We are reporting this case series because:

• Previously only case reports have been described and this is the first case series of BD

• Unusual presentations are less reported and easily misdiagnosed

• Our cases presented on unusual sites such as finger (second reported case), glans penis, and thigh (sun-protected sites)

• There was no evidence of immunosuppression or arsenic exposure in all cases

• Multiple pigmented variety type of presentation in one case (only one such case was reported in the literature)

Hence, a high index of suspicion, supported by histopathology, is required to diagnose and treat this condition without delay, which in turn may reduce the morbidity of patients.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Conflict of Interest

There are no conflicts of interest.

References

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2. Gahalaut P, Rastogi MK, Mishra N, Chauhan S. Multiple pigmented Bowen's disease: A diagnostic and therapeutic dilemma. Case Rep Oncol Med 2012; 2012: 342030
3. Neubert T, Lehmann P. Bowen's disease-a review of newer treatment options. Ther Clin Risk Manag 2008; 4: 1085-95
4. Quinn AG, Perkins W. Non melanoma skin cancer and other epidermal skin tumours. In: Burns DA, Breathnach SM, Cox NH, Griffiths CE. editors Rook's Textbook of Dermatology. Singapore: Wiley-Blackwell; 2010. 8. 32-4
5. Duncan KO, Geisse JK, Leffell DJ. Epithelial precancerous lesions. In: Wolff K, Goldsmith LA, Katz SI, Gilchrest BA, Paller BS, Leffell DJ. editors Fitzpatrick's Dermatology in General Medicine. New York: Mcgraw-Hill; 2008. 7. 1007-27
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7. Dandale A, Mantri MD, Thakkar V, Dhurat RS, Ghate S. et al. Bowen's disease: An unusual clinical presentation. Indian Dermatol Online J 2014; 5: 526-8
8. Yahya H, Mohammed A. Bowen's disease: Report of a case in a Nigerian man. West Afr J Med 2005; 24: 350-1
9. Papageorgiou PP, Koumarianou AA, Chu AC. Pigmented Bowen's disease. Br J Dermatol 1998; 138: 515-8
10. Ragi G, Turner MS, Klein LE, Stoll HLJr. Pigmented Bowen's disease and review of 420 Bowen's disease lesions. J Dermatol Surg Oncol 1988; 14: 765-9
11. Gong HS, Cho JH, Roh YJH, Chung MS, Baek GH. Bone invasion by squamous cell carcinoma in situ (Bowen's disease) of the finger during treatment with imiquimod 5% cream: Case report. J Hand Surg Am 2010; 35: 999-1002

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Publication History

Received: 29 July 2020

Accepted: 02 October 2020

Article published online:
14 May 2021

© 2020. Indian Society of Medical and Paediatric Oncology. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial-License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/.)

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